Long-gap esophageal atresia associated with foregut duplication – a case report and literature review

Kacper Kroczek, Katarzyna Lipa

Abstract


Coexisting long-gap esophageal atresia (EA) with a mediastinal duplication cyst is rare and difficult to diagnose. A 12-month-old boy, after multistage long-gap EA repair, presented with recurrent airway infections and a large mediastinal cystic lesion on computed tomography scans. Esophageal atresia repair was performed during infancy, with no coexisting pathology reported intraoperatively. Open Nissen fundoplication with gastrostomy and pyloroplasty was performed due to refractory gastroesophageal reflux. Recurrent airway infections were observed after an asymptomatic period. Thoracoscopic removal of the foregut duplication cyst was performed. At the 8-month follow-up, no recurrence of symptoms was reported. The coexistence of a foregut duplication cyst and EA is very rare, and diagnosis is challenging. Only 14 cases have been reported in the available literature. We recommend considering the possible association of EA and congenital foregut cyst intraoperatively, especially in symptomatic patients.

Keywords


esophageal atresia; esophageal duplication; long gap; case report; foregut cyst

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References


Lal DR, Gadepalli SK, Downard CD, Ostlie DJ, Minneci PC, Swedler RM, et al. Perioperative management and outcomes of esophageal atresia and tracheoesophageal fistula. J Pediatr Surg 2017;52(8):1245-51. doi: 10.1016/j.pedsurg.2016.11.046.

Gross RE. The surgery of infancy and childhood: its principles and techniques. Philadelphia, London: W. B. Saunders Company; 1953.

Spitz L. Oesophageal atresia. Orphanet J Rare Dis 2007;2:24. doi: 10.1186/1750-1172-2-24.

Shieh HF, Jennings RW. Long-gap esophageal atresia. Semin Pediatr Surg 2017;26(2):72-7. doi: 10.1053/j.sempedsurg.2017.02.009.

Stadil T, Koivusalo A, Pakarinen M, Mikkelsen A, Emblem R, Svensson JF, et al. Surgical repair of long-gap esophageal atresia: a retrospective study comparing the management of long-gap esophageal atresia in the Nordic countries. J Pediatr Surg 2019;54(3):423-8. doi: 10.1016/j.jpedsurg.2018.07.023.

Shalamon J, Schleef J, Höllwarth ME. Experience with gastro-intestinal duplications in childhood. Langenbecks Arch Surg 2000;385(6):402-5. doi: 10.1007/s004230000170.

Holcomb GW 3rd, Gheissari A, O’Neill JA Jr, Shorter NA, Bishop HC. Surgical management of alimentary tract duplications. Ann Surg 1989;209(2):167-74. doi: 10.1097/00000658-198902000-00006.

What are CARE guidelines? CARE. https://www.care-statement.org (12.11.2024).

Arbona JL, Fazzi JG, Mayoral J. Congenital esophageal cysts: Case report and review of literature. Am J Gastroenterol 1984;79(3):177-82.

McNally J, Charles AK, Spicer RD, Grier D. Mixed foregut cyst associated with esophageal atresia. J Pediatr Surg 2001;36(6):939-40. doi: 10.1053/jpsu.2001.23981.

Moore KL, Persaud TVN. The developing human: clinically oriented embryology. 8th ed. Philadelphia, PA: Saunders/Elsevier; 2008.

Qi BQ, Beasley SW, Williams AK. Evidence of a common pathogenesis for foregut duplications and esophageal atresia with tracheo-esophageal fistula. Anat Rec 2001;264(1):93-100. doi: 10.1002/ar.1125.

Azzie G, Beasley S. Diagnosis and treatment of foregut duplications. Semin Pediatr Surg 2003;12(1):46-54. doi: 10.1053/spsu.2003.50000.

Saha AK, Kundu AK. Tubular duplication of the oesophagus presenting with dysphagia. Singapore Med J 2014;55(6):e90-2. doi: 10.11622/smedj.2013261.

Horwitz JR, Lally KP. Bronchogenic and esophageal duplication cyst in a single mediastinal mass in a child. Pediatr Pathol Lab Med 1996;16(1):113-8.

Strollo DC, Rosado-de-Christenson ML, Jett JR. Primary mediastinal tumors: part II. Tumors of the middle and posterior mediastinum. Chest 1997;112(5):1344-57. doi: 10.1378/chest.112.5.1344.

Carachi R, Azmy A. Foregut duplications. Pediatr Surg Int 2002;18(5-6):371-4. doi: 10.1007/s00383-002-0835-y.

Bratu I, Laberge JM, Flageole H, Bouchard S. Foregut duplications: is there an advantage to thoracoscopic resection? J Pediatr Surg 2005;40(1):138-41. doi: 10.1016/j.jpedsurg.2004.09.025.

Nakahara K, Fujii Y, Miyoshi S, Yoneda A, Miyata M, Kawashima Y. Acute symptoms due to huge duplication cysts ruptured into the esophagus. Ann Thorac Surg 1990;50(2):309-11. doi: 10.1016/0003-4975(90)90761-t.

Narasimharao KL, Mitra SK. Esophageal atresia associated with esophageal duplication cysts. J Pediatr Surg 1987;22(11):984-5. doi: 10.1016/s0022-3468(87)80487-6.

Tapia RH, White VA. Squamous cell carcinoma arising in a duplication cyst of the esophagus. Am J Gastroenterol 1985;80(5):325-9.

Bower RJ, Sieber WK, Kiesewetter WB. Alimentary tract duplications in children. Ann Surg 1978;188(5):669-74. doi: 10.1097/00000658-197811000-00015.

Winslow RE, Dykstra G, Scholten DJ, Dean RE. Duplication of the cervical esophagus. An unrecognized cause of respiratory distress in infants. Am Surg 1984;50(9):506-8.

Galazka P, Skinder D, Styczynski J. Use of staged vs. primary repair in thoracoscopic esophageal atresia repair. J Pediatr Surg 2024;59(3):357-62. doi: 10.1016/j.jpedsurg.2023.10.055.

Galazka P, Skinder D, Styczynski J. Short-term and mid-term effects of thoracoscopic repair of esophageal atresia: No anastomotic leaks or conversions to open technique. Front Surg 2022;9:1009448. doi: 10.3389/fsurg.2022.1009448.

Snyder CL, Bickler SW, Gittes GK, Ramachandran V, Ashcraft KW. Esophageal duplication cyst with esophageal web and tracheoesophageal fistula. J Pediatr Surg 1996;31(7):968-9. doi: 10.1016/s0022-3468(96)90424-8.

Chuang MT, Barba FA, Kaneko M, Tierstein AS. Adenocarcinoma arising in an intrathoracic duplication cyst of foregut origin: a case report with review of the literature. Cancer 1981;47(7):1887-90.

Nobuhara KK, Gorski YC, La Quaglia MP, Shamberger RC. Bronchogenic cysts and esophageal duplications: common origins and treatment. J Pediatr Surg 1997;32(10):1408-13. doi: 10.1016/s0022-3468(97)90550-9.

Knod JL, Garrison AP, Frischer JS, Dickie B. Foregut duplication cyst associated with esophageal atresia and tracheoesophageal fistula: a case report and literature review. J Pediatr Surg 2013;48(5):E5-7. doi: 10.1016/j.jpedsurg.2013.02.071.

Spataru RI, Popoiu MC, Ivanov M. Foregut duplication cyst associated with esophageal atresia – one-stage neonatal surgical repair. Indian J Surg 2015;77(Suppl 1):52-5. doi: 10.1007/s12262-014-1122-6.

Escobar MA Jr, Welke KF, Holland RM, Caty MG. Esophageal atresia associated with a rare vascular ring and esophageal duplication diverticulum: a case report and review of the literature. J Pediatr Surg 2012;47(10):1926-9. doi: 10.1016/j.jpedsurg.2012.07.051.

Inan M, Basaran UN, Aksu B, Dereli M, Dortdogan Z. Esophageal duplication associated with esophageal atresia and tracheoesophageal fistula in a child. Int J Pediatr Otorhinolaryngol Extra 2007;2(3):202-5. doi: 10.1016/j.pedex.2007.05.010.

Yamagiwa I, Obata K, Ouchi T, Sotoda Y, Shimazaki Y. Heterotopic pancreas of the esophagus associated with a rare type of esophageal atresia. Ann Thorac Surg 1998;65(2):1143-4. doi: 10.1016/S0003-4975(98)00050-2.

Janik JS, Hernandez AM, Lilly JR. Esophageal atresia with proximal pouch tracheoesophageal fistula and noncommunicating esophageal duplication. Pediatr Surg Int 1988;3:70-3. doi: 10.1007/ BF00177086.

Santamaria JI, Nunez R, Sanjuan S, Serrano A. Esophageal atresia associated with cystic duplication of the esophagus. Chir Pediatr 1988;29(5):293-4.

Kirks DR, Filston HC. The association of esophageal duplication cyst with esophageal atresia. Pediatr Radiol 1981;11(4):214-6. doi: 10.1007/BF00972055.

Hemalatha V, Batcup G, Brereton RJ, Spitz L. Intrathoracic foregut cyst (foregut duplication) associated with esophageal atresia. J Pediatr Surg 1980;15(2):178-80. doi: 10.1016/s0022-3468(80)80012-1.




DOI: https://doi.org/10.21164/pomjlifesci.1136

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